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• W2944757191 startingPage "1601484" @default.
• W2944757191 abstract "Background: Spinal muscular atrophy type 1 (SMA1) is a devastating genetic disease for which gene-replacement therapy may bring substantial survival and quality of life benefits.Objective: This study investigated the cost-effectiveness of onasemnogene abeparvovec (AVXS-101) gene-replacement therapy for SMA1.Study design: A Markov model was used to estimate the incremental cost-effectiveness ratio (ICER), expressed as cost/quality-adjusted life year ($/QALY), of AVXS-101 versus nusinersen over a lifetime. Survival, healthcare costs and QALYs were estimated using natural history data for SMA patients who achieved motor milestones (sitting/walking). Health utility weights were obtained from the CHERISH trial.Setting: USA; commercial payer perspectiveParticipants: SMA1 infantsInterventions: AVXS-101 was compared to nusinersen.Main outcome measure: The primary outcome was the ICER.Results: Expected survival (undiscounted) over a lifetime predicted by the model was 37.20 life years for AVXS-101 and 9.68 for nusinersen (discounted QALYs, 15.65 and 5.29, respectively). Using a potential AVXS-101 price range ($2.5-5.0M/treatment), the average lifetime cost/patient was $4.2–6.6M for AVXS-101 and $6.3M for nusinersen. The ICER range was (-$203,072) to $31,379 per QALY gained for AVXS-101 versus nusinersen, indicating that AVXS-101 was cost-effective with prices of ≤$5M.Conclusion: Single-dose AVXS-101 was cost-effective compared to chronic nusinersen for SMA1 patients." @default.
• W2944757191 created "2019-05-16" @default.
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• W2944757191 date "2019-01-01" @default.
• W2944757191 modified "2023-10-16" @default.
• W2944757191 title "Cost-effectiveness analysis of using onasemnogene abeparvocec (AVXS-101) in spinal muscular atrophy type 1 patients" @default.
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• W2944757191 doi "https://doi.org/10.1080/20016689.2019.1601484" @default.
• W2944757191 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/6508058" @default.
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